Amy A Lemke, Katelyn C McNamara, R Jean Cadigan, Roselle Ponsaran, Aaron J Goldenberg, Carla A Rich, Candice R Finnila, Gregory M Cooper, Kyle B Brothers
{"title":"父母和社区护理专业人员如何使用基因诊断来通知护理:扩大效用的概念。","authors":"Amy A Lemke, Katelyn C McNamara, R Jean Cadigan, Roselle Ponsaran, Aaron J Goldenberg, Carla A Rich, Candice R Finnila, Gregory M Cooper, Kyle B Brothers","doi":"10.1007/s12687-026-00891-x","DOIUrl":null,"url":null,"abstract":"<p><p>Research on genomic testing outcomes for children with neurodevelopmental conditions often emphasizes clinical and personal utility, but rarely considers how a genomic diagnosis impacts care from non-physician community-based professionals such as those in private practice, early intervention, or school settings. Therefore, this study explored how caregivers and community care professionals use a genomic diagnosis to inform care. Semi-structured interviews were conducted with 23 caregivers of children with a genomic diagnosis and 30 community care professionals providing care to the child. Directed and summative content analysis was performed. Reported impacts included (1) improved care provision, (2) informed future planning, (3) enhanced social and community support, (4) enhanced understanding, (5) increased access to therapy-related services and equipment, (6) improved service quality and 5) elevated patient advocacy and engagement. Potential disutility arose from limited diagnostic information, provider knowledge gaps, insurance denials, and provider fear of treating rare conditions. A key neutral/mixed finding was that the diagnosis did not always impact care. Barriers to potential utility included financial burden, confusion about next steps, provider knowledge gaps, and lack of evidence-based guidance for rare diagnoses. Findings highlight that caregivers and community professionals may experience impacts from a genomic diagnosis (positive, negative, or neutral) that are understudied in empirical research, potentially because traditional concepts of utility fail to incorporate these domains. Broadening this conceptual framing could inform empirical research focused on identifying these effects and informing potential interventions. Further work is needed to characterize and quantify genomic test utility and disutility across diverse contexts.</p>","PeriodicalId":46965,"journal":{"name":"Journal of Community Genetics","volume":"17 3","pages":""},"PeriodicalIF":1.8000,"publicationDate":"2026-05-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13149733/pdf/","citationCount":"0","resultStr":"{\"title\":\"How parents and community care professionals use a genetic diagnosis to inform care: expanding the concept of utility.\",\"authors\":\"Amy A Lemke, Katelyn C McNamara, R Jean Cadigan, Roselle Ponsaran, Aaron J Goldenberg, Carla A Rich, Candice R Finnila, Gregory M Cooper, Kyle B Brothers\",\"doi\":\"10.1007/s12687-026-00891-x\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Research on genomic testing outcomes for children with neurodevelopmental conditions often emphasizes clinical and personal utility, but rarely considers how a genomic diagnosis impacts care from non-physician community-based professionals such as those in private practice, early intervention, or school settings. Therefore, this study explored how caregivers and community care professionals use a genomic diagnosis to inform care. Semi-structured interviews were conducted with 23 caregivers of children with a genomic diagnosis and 30 community care professionals providing care to the child. Directed and summative content analysis was performed. Reported impacts included (1) improved care provision, (2) informed future planning, (3) enhanced social and community support, (4) enhanced understanding, (5) increased access to therapy-related services and equipment, (6) improved service quality and 5) elevated patient advocacy and engagement. Potential disutility arose from limited diagnostic information, provider knowledge gaps, insurance denials, and provider fear of treating rare conditions. A key neutral/mixed finding was that the diagnosis did not always impact care. Barriers to potential utility included financial burden, confusion about next steps, provider knowledge gaps, and lack of evidence-based guidance for rare diagnoses. Findings highlight that caregivers and community professionals may experience impacts from a genomic diagnosis (positive, negative, or neutral) that are understudied in empirical research, potentially because traditional concepts of utility fail to incorporate these domains. Broadening this conceptual framing could inform empirical research focused on identifying these effects and informing potential interventions. Further work is needed to characterize and quantify genomic test utility and disutility across diverse contexts.</p>\",\"PeriodicalId\":46965,\"journal\":{\"name\":\"Journal of Community Genetics\",\"volume\":\"17 3\",\"pages\":\"\"},\"PeriodicalIF\":1.8000,\"publicationDate\":\"2026-05-07\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC13149733/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Journal of Community Genetics\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1007/s12687-026-00891-x\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q4\",\"JCRName\":\"GENETICS & HEREDITY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of Community Genetics","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1007/s12687-026-00891-x","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q4","JCRName":"GENETICS & HEREDITY","Score":null,"Total":0}
How parents and community care professionals use a genetic diagnosis to inform care: expanding the concept of utility.
Research on genomic testing outcomes for children with neurodevelopmental conditions often emphasizes clinical and personal utility, but rarely considers how a genomic diagnosis impacts care from non-physician community-based professionals such as those in private practice, early intervention, or school settings. Therefore, this study explored how caregivers and community care professionals use a genomic diagnosis to inform care. Semi-structured interviews were conducted with 23 caregivers of children with a genomic diagnosis and 30 community care professionals providing care to the child. Directed and summative content analysis was performed. Reported impacts included (1) improved care provision, (2) informed future planning, (3) enhanced social and community support, (4) enhanced understanding, (5) increased access to therapy-related services and equipment, (6) improved service quality and 5) elevated patient advocacy and engagement. Potential disutility arose from limited diagnostic information, provider knowledge gaps, insurance denials, and provider fear of treating rare conditions. A key neutral/mixed finding was that the diagnosis did not always impact care. Barriers to potential utility included financial burden, confusion about next steps, provider knowledge gaps, and lack of evidence-based guidance for rare diagnoses. Findings highlight that caregivers and community professionals may experience impacts from a genomic diagnosis (positive, negative, or neutral) that are understudied in empirical research, potentially because traditional concepts of utility fail to incorporate these domains. Broadening this conceptual framing could inform empirical research focused on identifying these effects and informing potential interventions. Further work is needed to characterize and quantify genomic test utility and disutility across diverse contexts.
期刊介绍:
The Journal of Community Genetics is an international forum for research in the ever-expanding field of community genetics, the art and science of applying medical genetics to human communities for the benefit of their individuals.
Community genetics comprises all activities which identify persons at increased genetic risk and has an interest in assessing this risk, in order to enable those at risk to make informed decisions. Community genetics services thus encompass such activities as genetic screening, registration of genetic conditions in the population, routine preconceptional and prenatal genetic consultations, public education on genetic issues, and public debate on related ethical issues.
The Journal of Community Genetics has a multidisciplinary scope. It covers medical genetics, epidemiology, genetics in primary care, public health aspects of genetics, and ethical, legal, social and economic issues. Its intention is to serve as a forum for community genetics worldwide, with a focus on low- and middle-income countries.
The journal features original research papers, reviews, short communications, program reports, news, and correspondence. Program reports describe illustrative projects in the field of community genetics, e.g., design and progress of an educational program or the protocol and achievement of a gene bank. Case reports describing individual patients are not accepted.